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      Treatment and follow-up of malignant struma ovarii: Regarding two cases

      case-report

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          Abstract

          Malignant struma ovarii (SO) is a rare tumor, and as a consequence, treatments and follow-up procedures are not clearly established. Presented in this study are two cases of suspicious ovarian masses, resected and corresponding to malignant SO on histopathology. Similar to thyroid cancer, we proposed complementary radioiodine therapy ( 131I) after total thyroidectomy (no malignancy was observed at this level in our two patients). Patients underwent treatment with 3.7 GBq 131I followed by post-therapy whole-body scintigraphy, which can detect residual disease or occult metastases. Thyroid remnant ablation increases the sensitivity and specificity of follow-up testing using serum thyroglobulin levels as a tumor marker. Our two patients remained disease-free for 3 and 5 years, respectively, after treatment.

          Highlights

          • Malignant struma ovarii is a rare ovarian tumor, corresponding to thyroid carcinoma.

          • It is generally diagnosed after surgical resection of an ovarian mass.

          • Iodine 131 therapy can be proposed for adjuvant therapy, after total thyroidectomy.

          • Follow-up must be extended for 20 years, using thyroglobulin level as a tumor marker.

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          Most cited references15

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          Clinical characteristics of struma ovarii.

          To evaluate the clinical characteristics of struma ovarii. Twenty-five cases of struma ovarii were reviewed retrospectively from June 1994 to April 2007. The presenting clinical, radiologic, and pathologic features of the patients were reviewed. The mean age of the patients in this study was 45.3 years. The majority was of premenopausal status. Sixteen patients had clinical symptoms such as low abdominal pain, palpable abdominal mass and vaginal bleeding. Although one patient had an abnormal thyroid function test, the laboratory findings normalized after operative treatment. CA-125 levels were elevated in 6 cases. Diagnosis by preoperative imaging studies were 8 dermoid cysts, while only 3 cases were diagnosed as struma ovarii. There were 4 cases of malignant struma ovarii, and no patients with recurrent disease. Struma ovarii is a rare tumor. The presented clinical, laboratory and radiological findings of patients are very diverse. The diagnosis was confirmed by pathologic findings. The treatment of benign struma ovarii is surgical resection only. The cases of malignant struma ovarii may need adjuvant treatment, but recurrence is uncommon.
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            Malignant struma ovarii: a case report and analysis of cases reported in the literature with focus on survival and I131 therapy.

            Malignant struma ovarii is a rare type of germ cell tumor that is most often diagnosed postoperatively. The natural history and optimal treatment regimen for the disease are essentially unknown due to the small numbers of published cases. A 32-year-old woman presented with pelvic pain and an ovarian mass that was ultimately treated by total abdominal hysterectomy/bilateral salpingo-oophorectomy. Postoperatively, she was diagnosed with a malignant struma ovarii. The patient was subsequently treated with thyroidectomy and I(131) ablation and is currently disease free. A Medline literature search was performed and clinical data from 23 additional cases were compiled. In this review of 24 cases, 16 patients were followed conservatively postoperatively while 8 received varied additional therapy (4 with I(131)). There were 8 recurrences and all occurred in the conservatively managed patients. I(131) for recurrent disease provided an initial complete response in 7 women. Treatment with thyroidectomy and I(131) should be considered in the first line of management for malignant struma ovarii.
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              Proliferative and histologically malignant struma ovarii: a clinicopathologic study of 54 cases.

              We reviewed 54 cases of struma ovarii with histologic features diverging from the normal pattern of benign thyroid tissue. These 54 lesions were divided into proliferative struma (41 cases) and malignant struma (13 cases). The patients diagnosed with proliferative struma ovarii ranged in age from 18 to 84 years (average, 44 years). The most common clinical findings among the proliferative struma patients were a mass (58%) and acute abdominal pain (12%). Preoperative evidence of hyperthyroidism was noted in three of the patients with proliferative struma, whereas one additional patient presented with ascites and hydrothorax ("pseudo-Meigs' syndrome"). Proliferative struma differed from the usual struma ovarii in that they comprised areas of densely packed follicles or papillary formations that raised the possibility of malignancy. However, none of the lesions that we have designated as proliferative struma ovarii showed histologic evidence of overlapping "ground glass" nuclei, vascular space invasion, or mitotic activity that would have supported an unequivocal diagnosis of malignancy. None developed metastases or recurrent disease. The 14 malignant struma ovarii manifested the classical features of thyroid carcinoma (including the presence of overlapping "ground glass" nuclei lining papillary formations and vascular space invasion). Patients with malignant struma ovarii ranged in age from 30 to 77 years (average, 50 years). Their clinical presentations included a mass (78%) and acute abdominal pain (22%). One patient had clinical and laboratory evidence of hyperthyroidism. On follow-up, one patient had persistent disease with peritoneal involvement, but distant metastases did not develop in any of these patients. A diagnosis of malignant struma ovarii should be reserved for lesions that exhibit the full range of changes seen in thyroid carcinoma arising in the cervical thyroid. By requiring that these rigid criteria be adhered to, the diagnosis of malignant struma ovarii will probably become less frequent as the more commonly encountered proliferative struma ovarii are recognized.
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                Author and article information

                Contributors
                Journal
                Gynecol Oncol Rep
                Gynecol Oncol Rep
                Gynecologic Oncology Reports
                Elsevier
                2352-5789
                08 June 2016
                August 2016
                08 June 2016
                : 17
                : 56-59
                Affiliations
                [a ]Department of Nuclear Medicine, Oscar Lambret Center, 3 rue Combemale, 59020 Lille Cedex, France
                [b ]Department of Surgery, Oscar Lambret Center, 3 rue Combemale, 59020 Lille Cedex, France
                [c ]Department of Biology, Unit of Morphological and Molecular Pathology, Oscar Lambret Center, 3 rue Combemale, 59020 Lille Cedex, France
                Author notes
                [* ]Corresponding author at: Department of Nuclear Medicine, Oscar Lambret Center, 3 rue Frederic Combemale, 59020 Lille Cedex, France.Department of Nuclear MedicineOscar Lambret Center3 rue Frederic CombemaleLille Cedex59020France a-oudoux@ 123456o-lambret.fr
                Article
                S2352-5789(16)30038-8
                10.1016/j.gore.2016.05.014
                4913172
                27355004
                0d360243-cd8b-479b-8a30-6d26c57b1797
                © 2016 The Authors

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 19 April 2016
                : 19 May 2016
                : 31 May 2016
                Categories
                Case Report

                malignant struma ovarii,radioactive iodine therapy,thyroid carcinoma

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